salmonella enterica serovar enteritidis brain abscess mimicking meningitis after surgery for glioblastoma multiforme a case report and review of the literature

C A S E R E P O R T Open AccessSalmonella enterica serovar Enteritidis brain abscess mimicking meningitis after surgery for glioblastoma multiforme: a case report and review of the liter

C A S E R E P O R T Open Access

Salmonella enterica serovar Enteritidis brain

abscess mimicking meningitis after surgery

for glioblastoma multiforme: a case report

and review of the literature

Léa Luciani1,2, Grégory Dubourg1,2, Thomas Graillon3, Estelle Honnorat4, Hubert Lepidi1, Michel Drancourt1,2, Piseth Seng1,2,4,5* and Andreas Stein1,2,4

Abstract

Background: Salmonella brain abscess associated with brain tumor is rare Only 11 cases have been reported to date Here we report a case of brain abscess caused by Salmonella enterica serovar Enteritidis mimicking

post-surgical meningitis in a patient with glioblastoma multiforme

Case presentation: A 60-year-old Algerian woman was admitted through an emergency department for a 4-day history of headache, nausea and vomiting, and behavioral disorders Surgery for cerebral tumor excision was

performed and histopathological analysis revealed glioblastoma multiforme On the seventh day post-surgery, she presented a sudden neurological deterioration with a meningeal syndrome, confusion, and fever of 39.8°C Her cerebrospinal fluid sample and blood cultures were positive for S enterica Enteritidis She was treated with ceftriaxone and ciprofloxacin On the 17th day post-surgery, she presented a new neurological disorder and

purulent discharge from the surgical wound Brain computed tomography revealed a large cerebral abscess

located at the operative site Surgical drainage of the abscess was performed and microbial cultures of surgical deep samples were positive for the same S enterica Enteritidis isolate She recovered and was discharged 6 weeks after admission

Conclusions: In this case report, a brain abscess was initially diagnosed as Salmonella post-surgical meningitis before the imaging diagnosis of the brain abscess The diagnosis of brain abscess should be considered in all cases

of non-typhoidal Salmonella meningitis after surgery for brain tumor Surgical brain abscess drainage followed by prolonged antibiotic treatment remains a major therapeutic option

Keywords: Brain abscess, Glioblastoma, Post-surgery meningitis, Salmonella, Salmonella enterica, MALDI-TOF,

Bacteria, Infection, Human

Background

Salmonella species are mainly known as common agents

of gastroenteritis worldwide Invasive Salmonella

in-fections have been reported due to their potential to

cause focal suppurative complications in urinary tract

infection, osteoarticular infection and liver abscess [1] Central nervous system Salmonella infection is rare and occurs primarily in young children [2] and immuno-compromised adults, including human immunodefi-ciency virus (HIV) infection and co-infected patients [3] and chronic granulomatous disease [4] Here, we report

a case of brain abscess caused by S enterica subspecies (subsp.) enterica serovar Enteritidis mimicking post-surgical meningitis in a patient with glioblastoma multi-forme We also review cases of Salmonella brain abscess

in patients with cerebral tumors

* Correspondence: sengpiseth@yahoo.fr

1 Aix Marseille Université, URMITE, UM63, CNRS 7278, IRD 198, Inserm 1095,

13005 Marseille, France

2 Pôle de Maladies Infectieuses, Hôpital de la Timone, Assistance Publique

Hôpitaux de Marseille, Institut Hospitalo-Universitaire Méditerranée Infection,

13005 Marseille, France

Full list of author information is available at the end of the article

© 2016 The Author(s) Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made The Creative Commons Public Domain Dedication waiver

Case presentation

In September 2015, a 60-year-old Algerian woman was

seen in the emergency department in Marseille, France for

a 4-day history of headache, nausea and vomiting, and

behavioral disorders She had an unremarkable medical

history apart from obesity (body mass index at 30.9 kg/

m2) Brain magnetic resonance imaging (MRI) revealed a

single 40×35 mm tumor in her right mesial temporal

region and a mass effect compression of her right lateral

ventricle with transtentorial herniation (Fig 1) She was

transferred to our neurosurgery department, where

leve-tiracetam and methylprednisolone led to neurological

improvement At that time, her leukocyte count was

elevated at 22×109/L (neutrophil count was 21×109/L,

platelet count was 291×109/L) Surgery for tumor removal

was performed on day 5 of her admission A histological

examination revealed glioblastoma multiforme (Fig 2) No

bacteria were seen on histological analysis

On the seventh day post-surgery, she presented a sudden

neurological deterioration with a meningeal syndrome,

confusion and fever of 39.8 °C Laboratory investigations

revealed an elevated leukocyte count at 13×109/L, elevated

neutrophils at 12.62×109/L, low lymphocytes at 0.15×109/L,

normal platelets at 154×109/L, and elevated C-reactive

protein at 304 mg/L Cerebrospinal fluid (CSF) sample

analysis revealed an elevated protein level of 2.93 g/L, a low

glucose level of 0.1 mmol/L, and a leukocyte count of 5400

cells/mm3 with 80 % neutrophils CSF cultures and blood

cultures were positive for S enterica The isolates from the

CSF and blood were further identified as S enterica subsp enterica serotype Enteritidis as identified by our national reference center for Salmonella (Institut Pasteur, Paris) The isolates were susceptible in vitro to amoxicillin, cef-triaxone, imipenem/cilastatin, gentamycin, co-trimoxazole and fluoroquinolone

A diagnosis of Salmonella meningitis was made and she was treated with ceftriaxone administered intra-venously 2 g/day and oral ciprofloxacin 500 mg every 8 hours On the 17th day post-surgery, she presented a new neurological disorder and purulent discharge from the surgical wound Brain computed tomography (CT) revealed a large cerebral abscess located at the operative site (Fig 3) Surgical drainage of the abscess was

Fig 1 Brain magnetic resonance imaging revealed a single 40×35 mm

tumor in the right mesial temporal region and a mass effect compression

of the right lateral ventricle with transtentorial herniation

Fig 2 A histological examination revealed glioblastoma multiforme without any microorganism identified on histological analysis

Fig 3 Computed tomography reveals a large cerebral abscess located at the operative site

Luciani et al Journal of Medical Case Reports (2016) 10:192 Page 2 of 5

diagnosis of brain abscess

corticosteroid treatment Our case (2015) 60 years, female,

Algeria

Yes, multiforme glioblastoma

deterioration, meningeal syndrome

(CSF, blood, pus, brain abscess)

Good

Rodriguez, Valero, and

Watanakunakorn 1986 [8]

28 years, male, Ohio (USA)

Yes, metastatic carcinoma

papilledema

(radiotherapy)

Salmonella Enteritidis (brain tissue and blood)

Good

Sharma, Raja, and

Shivananda 1986 [9]

32 years, male, India

Yes, malignant astrocytoma

somnolence

duration

Noguerado et al 1987 [ 10] 78 years, male,

Spain

Yes, multiforme glioblastoma

deteriorated, fever, meningeal syndrome, septic shock

(CSF and blood)

Died

Bossi et al 1993 [ 11] 24 years, male,

Tunisia

Yes, multiforme glioblastoma

duration

Salmonella Enteritidis (CSF, blood and brain abscess)

Good

Shanley and Holmes

1994 [12]

28 years, female, Hawaii (USA)

Yes, craniopharyngioma

mentioned

Sudden loss of vision Yes,

Hypophysectomy

to decompress optic chiasm

Not mentioned Salmonella Typhi (pus,

brain abscess)

Good

Fiteni et al 1995 [ 13] 49 years, female,

France

(CSF, blood and brain abscess)

Residual hemiparesis

Sarria, Vidal, and

Kimbrough Iii 2000 [14]

58 years, female, Texas (USA)

Yes, multiforme glioblastoma

syndrome, hemiparesis, coma

and local application

Salmonella Enteritidis (material)

Died

Kumari and Kan 2000 [15] 59 years, male,

Washington (USA)

Yes, metastatic adenocarcinoma

confusion

(cerebral abscess)

Good

Schröder et al 2003 [ 16] 46 years, female,

Germany

Yes, craniopharyngioma

craniotomy site

not known Salmonella Enteritidis

(pus, brain abscess)

Coxitis abscess Aissaoui et al 2006 [ 17] 72 years, male,

Morocco

Yes, oligodendroglioma

deterioration

patient died

Salmonella Enteritidis (CSF and blood)

Died

Sait et al 2011 [ 18] 57 years, male,

not known

Yes, multiforme glioblastoma

wound, meningeal signs

(material and blood)

Good CSF cerebrospinal fluid

performed by craniotomy, which confirmed the

diag-nosis of intraparenchymal abscess located at the

glio-blastoma resection site Microbial cultures of surgical

deep samples were positive for S enterica subsp enterica

serovar Enteritidis, which were susceptible to all

anti-biotics tested above She was discharged 6 weeks after

admission Prolonged 10-day anaerobic bacterial cultures

of her CSF, bloodstream and brain abscess were

nega-tive A combination of ceftriaxone-ciprofloxacin was

given for 6 weeks, and ciprofloxacin treatment was

prolonged for 3 months because of the infectious risk

due to chemotherapy immunosuppression No

neuro-logical sequelae were noted Evaluation of the immune

system remained normal and HIV serology was negative

Discussion

Here we report a case of brain abscess due to S enterica

subsp enterica serovar Enteritidis mimicking meningitis

occurring after surgery for glioblastoma Salmonella

brain abscesses are rarely reported Only a few cases of

typhoidal Salmonella brain abscess have been reported

in immunocompetent adults, usually related to situations

promoting their incidence, including recent travel in

endemic areas [5], typhoid fever [6], or ingestion of

contaminated milk [7] To the best of our knowledge,

only 11 cases of Salmonella brain abscess associated

with brain tumor have been reported [8–18] Most of

these cases (nine cases) were caused by non-typhoidal

Salmonella, including eight cases of S enterica

Enteriti-dis and one case of Salmonella enterica Typhimurium

However, S enterica Typhimurium is usually responsible

for invasive human salmonellosis [19] Glioblastoma is

the main type of brain tumor that has been associated

with Salmonella brain abscess (four cases), and all of

these cases were caused by S enterica Enteritidis

(Table 1)

Symptoms of Salmonella brain abscess associated with

brain tumor are heterogeneous Most cases (six cases)

have occurred after surgical resection of a brain tumor,

initially indicated by fever or neurological deterioration

and confusion However, meningeal signs were noted in

three reported cases In our case, the brain abscess was

initially diagnosed as Salmonella post-surgical meningitis

before imaging diagnosis of the brain abscess In our

case, the diagnosis of glioblastoma multiforme was

suggested by brain MRI and confirmed by a histological

examination of the surgical biopsy In vivo imaging

technology, such as molecular imaging, is useful in the

diagnosis of brain tumors [20] and might be helpful to

differentiate bacterial abscess from tumoral tissues and

underlying primary disease [21]

In the literature, Salmonella species have been

iden-tified in purulent exudates from brain abscesses (six cases)

and in blood cultures (six cases) and CSF cultures (four

cases) In our case, Salmonella isolates were identified in the blood, CSF and brain abscess Most cases in the lite-rature were treated with systemic corticosteroids for brain tumor (eight cases) when the Salmonella brain abscess was diagnosed The prognosis is relatively good with antibiotic treatment There is no comparative study on the use of dual antibiotic therapy rather than single antibiotic for this indication Nevertheless, we decided

to treat our case initially with a 6-week combination

of ceftriaxone-ciprofloxacin due to a significant risk of immunosuppression related to treatment of the glio-blastoma multiforme and the large brain abscess The duration of antibiotic treatment in the literature varied from 4 weeks to 3 months Most cases in the litera-ture (nine cases) were treated surgically for the brain abscess However, three patients died and two patients had complications, including residual hemiparesis in one case and a hip abscess in one case

Chronic carriage of Salmonella, primarily biliary, may persist after infection (about 1 % of cases) [22] In our case, septic signs and digestive symptoms such as gastro-enteritis were absent on admission and the clinical symptoms of brain abscess such as fever, meningeal signs, and neurological deterioration occurred only at 1 week post-surgery for glioblastoma These phenomena might be explained by Salmonella’s tropism for necrotic tissue [23], and the central nervous system infection could be secondary to blood dissemination of Salmo-nella from digestive reservoirs in the bile or intestine Unfortunately, this hypothesis is difficult to confirm due

to the transitory carriage and because a stool culture had unfortunately not been performed

Conclusions Salmonella brain abscess is rare but can occur in appa-rently immunocompetent adult patients with brain tumor The diagnosis of brain abscess should be consi-dered in all cases of non-typhoid Salmonella meningitis after surgery for brain tumor Prolonged antibiotic treat-ment after surgical brain abscess drainage remains a major therapeutic option

Acknowledgements

We thank the house officers and medical staff for their confidence in our management of the patient.

The authors have no relevant affiliations or financial involvement with any organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript No assistance was utilized in the writing of this manuscript.

Availability of data and supporting materials Medical imaging data will not be shared because it is not fully anonymous.

Authors ’ contributions LL: 1st author, clinical data collection; he was involved in drafting the manuscript GD: 2nd author, microbiological data collection, manuscript revision TG: 3rd author, clinical data verification and manuscript revision EH: 4th author; surgical data verification and manuscript revision HL: 5th Luciani et al Journal of Medical Case Reports (2016) 10:192 Page 4 of 5

author, histological analysis, manuscript revision, and discussion section.

MD: 6th author, microbiological data verification and manuscript revision.

PS: first final author and corresponding author; he made substantial

contributions to study conception and design, clinical data verification,

discussion section, and manuscript revision AS: second final author, clinical

data verification, discussion section, and final approval of the version to be

published All authors read and approved the final manuscript.

Competing interests

The authors declare that they have no competing interests.

Consent for publication

Written informed consent was obtained from the patient for publication of

this case report and any accompanying images A copy of the written

consent is available for review by the Editor-in-Chief of this journal.

Author details

1

Aix Marseille Université, URMITE, UM63, CNRS 7278, IRD 198, Inserm 1095,

13005 Marseille, France 2 Pôle de Maladies Infectieuses, Hôpital de la Timone,

Assistance Publique Hôpitaux de Marseille, Institut Hospitalo-Universitaire

Méditerranée Infection, 13005 Marseille, France 3 Service de neurochirurgie,

Hôpital de la Timone, Assistance Publique Hôpitaux de Marseille, 13005

Marseille, France 4 Service des Maladies Infectieuses, Hôpital de la

Conception, 147, boulevard Baille, 13005 Marseille, France 5 Unité de

Recherche sur les Maladies Infectieuses et Tropicales Emergentes, Faculté de

Médecine, Aix Marseille Université, 27, Boulevard Jean Moulin, 13385

Marseille, Cedex 5, France.

Received: 9 March 2016 Accepted: 3 June 2016

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